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      A selective IgA deficiency in a boy who presented recurrent parotitis

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          Abstract

          Recurrent parotitis is a non-obstructive, non-suppurative inflammatory disease which is characterized by unilateral or bilateral parotid gland swelling attacks. It is also known as juvenile recurrent parotitis. Although the etiology is unknown, congenital malformations of the ductus, genetic predisposition, infections, allergies, autoimmune diseases, and some immune deficiencies are blamed. Here, we present a case report of recurrent parotitis with selective immunoglobulin A deficiency in a six-year-old boy. The patient was presented to us with a new episode of swelling of left parotid region. In the last 2 years, the patient suffered from recurrent parotitis which lasted for approximately 5 days in ten individual episodes.

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          Most cited references 15

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          Childhood common variable immunodeficiency with autoimmune disease.

          Clinical and laboratory findings in eight patients with childhood common variable immunodeficiency and autoimmune disease are described. Six of the eight patients had initial signs of the disease, persistent secretory diarrhea, recurrent upper respiratory tract infections, or both, in the first year of life. Autoimmune manifestations included idiopathic thrombocytopenia (4/8), hemolytic anemia (3/8), secretory diarrhea (4/8), arthritis (2/8), chronic active hepatitis (2/8), parotitis (2/8), and Guillain-Barré syndrome (2/8). In addition to the expected sinusitis, otitis, and pneumonia caused by encapsulated bacteria, these patients also had severe infections with viruses of the herpes group. Most of these patients had lymphadenopathy, splenomegaly, growth failure, and failure to develop secondary sexual characteristics. Laboratory studies demonstrated a significant increase in the ratio of T cells expressing the T helper phenotype (OKT4) to T cells expressing the T suppressor-cytotoxic phenotype (OKT8) (T4/T8). This increase could be attributed to a decrease in the absolute number of T8 cells. Additional findings included fluctuating levels of serum immunoglobulins and markedly diminished in vitro antibody production by B cells. The clinical course was relapsing and remitting, and dominated by the autoimmune manifestations of the disease. This group of patients constitutes a distinct subset of children with hypogammaglobulinemia, a subset with a complex, multisystemic disorder associated with significant morbidity and mortality.
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            Recurrent parotitis of childhood.

            Recurrent parotitis (RP) of childhood is a rare condition of unknown aetiology, probably immunologically mediated. To review the clinical presentation, diagnosis and management of RP of childhood. Retrospective study from 1983 to 2004 of children diagnosed with RP of childhood at a tertiary children's hospital. We identified 53 children, 37 (70%) male and 16 (30%) female. The age of onset was biphasic, with peaks at 2-5 years of age and at 10 years. The commonest symptoms were swelling (100%), pain (92.5%) and fever (41.5%). Symptoms usually lasted 2-7 days with a median of 3 days. The mean frequency was 8 episodes per year. The diagnosis was often delayed, >1 year in 70% of patients, maximum 8 years. The most common diagnoses, before the definitive diagnosis of RP, were mumps (21%), 'infection' (15%) and stones (11%). Sialogram (57%) and/or ultrasound (41%) showed sialectasis in 81% of patients. Over half the patients (54%) were given antibiotics at least once to treat the parotitis. Two children had hypogammablobulinaemia, one child had human immunodeficiency virus infection, and one child had Sjogren's syndrome. Two children had high titre antinuclear antibodies. Recurrent parotitis had a biphasic age distribution. The major clinical features that distinguish it from other causes of parotid swelling are the lack of pus and recurrent episodes. A clinical diagnosis can often be confirmed by ultrasound. Antibiotics do not have a role in treatment. Affected children should be screened for Sjogren's syndrome and immune deficiency.
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              Natural history of patients with recurrent parotitis and punctate sialectasis.

              The clinical features of 68 patients (26 children and 42 adults) who suffered recurrent attacks of parotitis and in whom sialography had revealed punctate sialectasis of the affected gland is described. The sex incidence was equal in those patients in whom symptoms commenced during childhood (younger than 15). When symptoms commenced later in life, however, there was a marked preponderance of females (female:male = 7.5:1). Bilateral sialography in 16 patients with unilateral symptoms revealed punctate sialectasis in the asymptomatic gland in 11 (69 per cent) of patients. Neither the presence of the sicca syndrome nor auto-antibodies had a significant predictive value as to the outcome of the disease. Five-year follow-up of 52 patients revealed that 56 per cent of adults and 64 per cent of children had shown spontaneous improvement of symptoms with symptomatic treatment alone. In 40 per cent of adults and 4 per cent of children, however, the persistence or worsening of symptoms necessitated parotidectomy. We believe that these results of conservative management indicate that, at least in the first instance and particularly in children, conservative management is justified and that the use of radiotherapy or steroids (with their attendant morbidity) is unnecessary.
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                Author and article information

                Journal
                1886
                122234
                European Journal of Microbiology and Immunology
                EuJMI
                Akadémiai Kiadó, co-published with Springer Science+Business Media B.V., Formerly Kluwer Academic Publishers B.V.
                2062-509X
                2062-8633
                1 June 2014
                : 4
                : 2
                : 144-146
                Affiliations
                [ 1 ] Department of Pediatric Immunology, Erciyes University School of Medicine, Kayseri, Turkey
                [ 2 ] Department of Pediatrics, Erciyes University School of Medicine, Kayseri, Turkey
                Author notes

                Supplementary material is available for this article at 10.1556/EuJMI.4.2014.2.8 and is accessible for authorized users.

                [* ] +90 352 207 66 66/25300, +90 352 437 58 25, himmetakar@ 123456gmail.com
                Article
                8
                10.1556/EuJMI.4.2014.2.8
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                Case Study

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